European Journal of Echocardiography 2008 9(1):163-166; doi:10.1016/j.euje.2007.06.020
Published on behalf of the European Society of Cardiology. All rights reserved. © The Author 2007. For permissions please email: journals.permissions@oxfordjournals.org
Giant septal cavity due to coronary artery fistula and ventricular septal dissection after cardiac surgery
Luísa M. Branco*,
Joana Feliciano,
Duarte Cacela,
Ana Galrinho,
Rita M. Fernandes,
Carlos S. Salomão,
José M. Gonçalves and
Rui C. Ferreira
Department of Cardiology, Santa Marta Hospital, Rua de Santa Marta, Lisbon, Portugal
Received 24 February 2007; accepted after revision 20 June 2007; online publish-ahead-of-print 24 September 2007.
* Corresponding author. Tel: +351 917644152; fax: +351 8465469. E-mail address: saciv{at}netcabo.pt
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Abstract
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Ventricular septal dissection may rarely result from infective
endocarditis, myocardial infarction or sinus of Valsalva aneurysm
progression. A rare case that developed in a 66-year-old female
after mitral valve replacement for severe mitral regurgitation
with cordal rupture is presented and discussed. It resulted
from a coronary artery fistula, from a septal branch, to a 6
cm wide saccular cystic cavity within the interventricular septum,
which was detected by transthoracic echocardiography some months
after surgery. Coronary arteries were anatomically normal. Coronary
angiography was helpful to clarify the origin of the fistula.
Later on the patient developed heart failure and atrial fibrillation,
but she refused any further intervention. She has been followed
up for more than 7 years and is presently stable on medical
therapy.
A review of the peculiar aspects of the case is done and the most relevant aspects and images are presented and discussed.
Keywords: Septal dissection; Pseudoaneurysm; Transthoracic echocardiography; Transesophageal echocardiography; Coronary arteriovenous fistula; Cardiac surgery
Ventricular septal dissection may occur rarely after aortic
valve endocarditis with spread of the infection through the
septal tissues or after myocardial infarction with irregular
septal disruption before ventricular septal rupture and shunt
development.
1 It may also be a complication of chest trauma
2 or sinus of Valsalva aneurysm.
3 Coronary artery fistula (CAF)
may be congenital
4,5 or occur after chest trauma,
6 or endomyocardial
biopsy (
7).Very rarely they may result from injury at the time
of heart surgery with myectomy.
8,9
A very unusual case of septal dissection with a very large pseudoaneurysm formation in connection with a septal branch fistula occurred at Santa Marta Hospital after mitral valve replacement. Due to its peculiar characteristics and evolution it was decided to report it and discuss its particular aspects.
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Case report
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A 65-year-old female patient in NYHA class III had, in March
2000, her mitral valve replaced by a Carbomedics 29 prosthesis.
She had severe mitral regurgitation, with cordal rupture and
flail posterior mitral valve leaflet, severe pulmonary artery
hypertension (80 mmHg), slightly depressed left ventricular
function, grade II/IV tricuspid regurgitation, normal coronary
arteries and persistent atrial fibrillation. Before surgery
transthoracic echocardiography (TTE) and transesophageal echocardiography
(TEE) had been performed. The perioperative course was uneventful.
She was discharged on digoxin, lisinopril, hydrochlorothiazide
and aminophylline (as she had chronic bronchitis). The prosthetic
sounds were normal and no murmurs were heard on auscultation.
Pulmonary auscultation showed only some wheezes and ronchi.
She improved clinically, becoming NYHA class I and remained
in sinus rhythm with amiodarone.
A routine TTE performed in November 2002 showed a very large cystic cavity within the interventricular septum (IVS), measuring 53x53 mm. It filled in diastole from the IVS near the left ventricular border and a faint systolic flow was detected at the right ventricular border. Its size increased later to 53x64 mm (Figures 1–3, video clips 1 and 2). TEE was also performed and confirmed a large 45x55.5 mm septal cavity with spontaneous contrast and no thrombus inside. The left atrium was slightly enlarged (40 mm) and the left ventricular cavity was in the upper limits of normal. She was submitted to cardiac magnetic resonance, which showed a round cavity in the medial and apical septum measuring 5x5 cm, with blood inside, but no thrombus, suggestive of pseudoaneurysm (Figure 4).
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Figure 1 Transthoracic parasternal long axis view echocardiogram (upper image) and reverse long axis view (lower image) showing a large cystic cavity within the interventricular septum, measuring 5.21x5.55 or 5.11x6.21 cm, respectively. PS, pseudoaneurysm; RV, right ventricle; LA, left atrium; LV, left ventricle.
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Figure 2 Transthoracic long axis parasternal view (lower image) showing a red flow entering the cystic anterior cavity. In the upper image a colour M mode study demonstrates that the flow is diastolic, consistent with coronary like flow. PS, pseudoaneurysm; AO, aorta; LA, left atrium; LV, left ventricle; IVS, interventricular septum.
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Figure 3 Apical four-chamber transthoracic echocardiogram showing a 5.15x4.75 cm cavity within the mid and apical interventricular septum bulging into the ventricular cavities. In the middle image one can see a diastolic flow entering the cavity from the left. The lower panel shows a triplane view of the apical pseudoaneurysmal cavity. PS, pseudoaneurysm; RV, right ventricle; RA, right atrium; LA, left atrium; LV, left ventricle; P, prosthesis.
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In 2003 she was hospitalized and coronary arteries were morphologically
normal. A fistula from a septal coronary artery branch draining
into a very large cavity within the septum was identified (
Figure 5).
No significant shunt was detected and pulmonary artery systolic
pressure was 40 mm Hg and capillary wedge pressure was 10 mmHg.
Surgery or percutaneous closure of the fistula, namely with
coils or with a left anterior coronary artery stent, that would
occlude the entry into the septal branch, were proposed, but
the patient refused them. She remained in NYHA class II, with
a typical chest pain. The ECG showed sinus rhythm with inverted
T waves in V2–V5 and trivial ST depression in V4–V5.
Amiodarone was later changed into verapamil and digoxin. A nuclear
scan was also performed and showed a very large septal, apical
anterior and inferior juta-apical perfusion defect, that did
not improve with nitrates. Serial TTE showed always a normally
functioning mitral valve prosthesis (peak velocity 1.18 m/s,
mean gradient 2.4 mmHg).
In October 2004 she had a deterioration of her clinical condition
with entry into atrial fibrillation. The patient refused electrical
cardioversion. In May 2006 she deteriorated clinically, becoming
NYHA class III and needed to be hospitalized in congestive heart
failure. She was treated with intravenous diuretic and vasodilators
and improved markedly.
She is now 72 years old, in NYHA class II, leading a practically normal life, on acenocumarol, lisinopril, furosemide, spironolactone, allopurinol and bronchodilators. Her left and right ventricular dimensions have remained stable and the septal cavity has increased only slightly. During the 7 years of follow up she had only one episode of serious clinical deterioration. She continues to refuse other means of treatment for her condition.
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Discussion
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A very large septal pseudoaneurysm formation is reported, which
resulted from a CAF that developed after cardiac valve surgery.
The patient was at first almost asymptomatic, but later developed
heart failure mostly due to a steal effect and cardiac chamber
compression by the giant septal cavity. A continuous murmur
may be present and is highly suggestive of a CAF,
10 but it did
not exist in this patient. Coronary angiography was very important
in this case to clarify the correct etiology of the cystic septal
cavity. Treatment is advocated for symptomatic patients and
for those asymptomatic who are at risk for future complications.
10 Possible therapeutic options include surgical correction or
transcatheter closure.
10
Acquired CAF occurring after cardiac surgery have rarely been reported, and are usually anecdotal cases related to myectomy8 in congenital heart disease or in hypertrophic cardiomyopathy.9 Chiu et al. reported 10 CAF detected by echocardiography after open heart surgery for congenital heart disease.8 Their incidence was 0.44%.8 The incidence of CAF detected during diagnostic coronary angiography is also very low.4,5 Vavuranakis found a 0.1% incidence among 33 600 patients submitted to diagnostic cardiac catheterization and we found 0.28% in 5000 similar patients.4,5 Their prognosis in adults seems to be good.4,5,10
IVS dissection is a rare condition, which can uncommonly complicate an AMI.1 Mariscalco et al. have recently reported a case of unruptured IVS dissection observed after AMI and diagnosed by TTE.1 An echo-free space within the thickness of IVS, extended from the apex to the mid-portion, for a total length of about 30 mm,1 very similar, though smaller, to this case. The predischarge contrast echocardiographic examination (Levovist) demonstrated echogenicity increase of the neocavitation, with partially organised thrombi,1 which this patient never demonstrated, probably due to the anticoagulation she needs because of a mechanical prosthesis. IVS dissection with or without left ventricular communication may also be a rare complication of a sinus of Valsalva aneurysm, infective endocarditis or chest trauma.2,3
In this patient there were no previous AMI, septal myectomy, chest trauma, endocarditis or endomyocardial biopsy. The previous coronary angiogram was normal and TTE and TEE performed before surgery had not demonstrated any septal cavity. Septal disruption might have resulted from a septal puncture to evaluate intracavitary pressures. Marek et al. reported a case of postoperative iatrogenic left coronaroventricular fistula11 demonstrated by TTE 6 years after aortic valve replacement. They advocated that the CAF was created during perioperative LV purging and the implantation of a needle through the right ventricle and IVS.11 A similar etiology might explain our case as well, although to our knowledge there is no similar case in the literature.
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Supplementary material
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Supplementary material associated with this article can be found in the online version.
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Acknowledgments
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We thank Dr. Pedro Matos for having performed the cardiac magnetic
resonance and Dr. João Calqueiro for having performed
the nuclear scan of the patient.
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References
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- Mariscalco G, Blanzola C, Leva C, Cattaneo P, Mantovani V, Ferrarese S, et al. Unruptured ventricular septal wall dissection. A case report. J Cardiovasc Surg (Torino) (2006) 47:349–52.[Medline]
- Stajer D, Kariz S. Ventricular septal rupture following blunt chest trauma after a long delay: a case report. Int J Cardiol (1994) 47:187–8.[CrossRef][Web of Science][Medline]
- Engel PJ, Held JS, van der Bel-Kahn J, Spitz H. Echocardiographic diagnosis of congenital sinus of Valsalva aneurysm with dissection of the interventricular septum. Circulation (1981) 63:705–11.[Abstract/Free Full Text]
- Branco L, Agapito A, Ramos J M, Patrício L, Monteiro I, Galrinho A, et al. Fístulas coronárias – revisão clínica e angiogáfica. Rev Port Cardiol (1994) 13:243–52.[Medline]
- Vavuranakis M, Bush CA, Boudoulas H. Coronary artery fistulas in adults: incidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn (1995) 35:116–20.[Web of Science][Medline]
- Collet F, Bru P, Jouve B, Cointe R, Barragan P, Metge M, et al. Post traumatic fistula of the left anterior descending coronary artery and left ventricle. A propos of a new case. Arch Mal Coeur Vaiss (1990) 83:267–70.[Web of Science][Medline]
- Dubel HP, Romaniuk P, Warnke H. Coronary ventricular fistulas in patients with heart transplants. Herz (1991) 16:55–9.[Web of Science][Medline]
- Chiu SN, Wu MH, Lin MT, Wu ET, Wang JK, Lue HC. Acquired coronary artery fistula after open heart surgery for congenital heart disease. Int J Cardiol (2005) 103:187–92.[CrossRef][Web of Science][Medline]
- Awasthi A, Wormer D, Heggunje PS, Obeid A. Long-term follow-up of acquired coronary artery fistula after septal myectomy for hypertrophic cardiomyopathy. J Am Soc Echocardiogr (2002) 15:1104–7.[CrossRef][Web of Science][Medline]
- Gowda RM, Vasavada BC, Khan IA. Coronary artery fistulas: clinical and therapeutic considerations. Int J Cardiol (2006) 107:7–10.[CrossRef][Web of Science][Medline]
- Marek A, Rey JL, Tribouilly C, Jarry G, Avinee P, Lesbre JP, et al. Postoperative iatrogenic left coronaoventricular fistula. Demonstration by color two-dimensional Doppler. Arch Mal Coeur Vaiss (1991) 84:419–23.[Web of Science][Medline]
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